Chronic pain and fatigue in multiple osteochondroma and Ollier disease, a systematic review.
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INTRODUCTION: Multiple osteochondromas (MO) and Ollier disease are characterized by multiple tumours in the skeletal bones. They lead to bone deformity, restricted joint motion, interference with skeletal growth and increased risk of malignant transformation. The aim of this systematic review is to increase knowledge on chronic pain and fatigue in these diagnoses and identify gaps and topics for future research. MATERIALS AND METHODS: A systematic literature search was conducted in various databases in 2022 and updated in 2023. All types of studies were included that reported data on chronic pain and/or fatigue/vitality in patients with either multiple osteochondromas or Ollier disease. Study quality was assessed with Joanna Briggs Institute JBI's critical appraisal tools. RESULTS: A total of 63 studies (55 on multiple osteochondromas, 7 on Ollier disease, 1 on both), with a total of 1252 participants was found. The majority were case reports. No RCT was found. Fifty-six of 63 studies reported data only on pain; seven reported data on both pain and fatigue/vitality. Out of 63 studies, 29 used standardized assessment methods for pain, and seven for fatigue. Prevalence for chronic pain (when reporting non-site-specific pain) was reported as ranging from 80% - 95% for adults and 54-64% for children with multiple osteochondromas. Studies reporting fatigue showed a higher prevalence of severe fatigue or lower vitality scores when compared to other patient groups or the normal population. Pain and fatigue impacted ADL, mood, sleep, social and physical activities. Different interference factors for pain and fatigue were reported. Treatment strategies included "wait-and-see", surgery, physiotherapy, and medication. CONCLUSIONS: Chronic pain and fatigue appear to be an important complaint in patients with multiple osteochondromas impacting the patient's life. There is limited evidence on this topic for patients with Ollier disease. More research, particularly with larger cohort studies, is needed to be able to identify causation and associated factors, define optimal treatment and better understand the impact on patients' life and their coping strategies. TRIAL REGISTRATION: PROSPERO CRD42022379329.